Khalilian Sheyda, Fathi Mohadeseh, Tangestani Raheleh, Larki Pegah, Sayad Arezou, Ghafouri-Fard Soudeh, Miryounesi Mohammad
Student Research Committee, School of Medicine, Shahid Beheshti University of Medical Sciences, Tehran, Iran.
Department of Medical Genetics, Shahid Beheshti University of Medical Sciences, Tehran, Iran.
Hum Genomics. 2025 May 13;19(1):54. doi: 10.1186/s40246-025-00771-4.
Limb-girdle muscular dystrophies (LGMD) designate diverse types of muscular dystrophies that predominantly affect proximal skeletal muscles. Although both autosomal recessive and dominant forms exist, the majority of cases are inherited in an autosomal recessive manner. Since the spectrum of genetic variants that cause this disorder is quite broad, next-generation sequencing techniques are the best diagnostic tools for LGMD. In this study, we provide an overview of mutation spectrum of LGMD-related genes in the Iranian patients using whole exome sequencing. Notably, CAPN3 and LAMA2 genes were the genes encompassing the highest frequencies of pathogenic or likely pathogenic variants in this cohort. Pathogenic and likely pathogenic variants were identified in CAPN3 gene in total of 10 cases out of 48 cases tested (20%). In addition, different variants in each of POMGNT1 and TTN genes were detected in five and four patients, respectively. Three patients had DYSF variants (6%). While the inheritance of the majority of cases was supposed to be in an autosomal recessive manner, in three cases, the disease inheritance was best explained by the dominant type (c.947 C > T variant in the DNAJB6, c.746G > A variant in the LMNA, and c.1417G > A variant in the TNPO3). The current study broadens the spectrum of LGMD-related mutations among Iranian patients and facilitates genetic counseling in the affected families.
肢带型肌营养不良症(LGMD)指的是多种主要影响近端骨骼肌的肌营养不良症。虽然存在常染色体隐性和显性两种形式,但大多数病例是以常染色体隐性方式遗传的。由于导致这种疾病的基因变异谱相当广泛,下一代测序技术是诊断LGMD的最佳工具。在本研究中,我们使用全外显子组测序概述了伊朗患者中LGMD相关基因的突变谱。值得注意的是,在该队列中,CAPN3和LAMA2基因是包含致病性或可能致病性变异频率最高的基因。在48例检测病例中,共有10例(20%)在CAPN3基因中鉴定出致病性和可能致病性变异。此外,分别在5例和4例患者中检测到POMGNT1和TTN基因中的不同变异。3例患者有DYSF变异(6%)。虽然大多数病例的遗传方式被认为是常染色体隐性,但在3例病例中,疾病遗传最好用显性类型来解释(DNAJB6中的c.947 C>T变异、LMNA中的c.746G>A变异和TNPO3中的c.1417G>A变异)。本研究拓宽了伊朗患者中LGMD相关突变的谱,并有助于为受影响家庭提供遗传咨询。